Hospitalization- and death-related financial and employment effects in parents of children with life-limiting conditions: a fixed-effects analysis

The purpose of this study is to investigate out-of-pocket non-medical expenses and employment-related outcomes in families of children with life-limiting conditions, specifically, to quantify the financial and employment implications of two events: a child’s hospitalization and death. This cohort study used panel data collected prospectively for a larger study investigating the effectiveness of specialized pediatric palliative care. Participants were recruited by medical professionals between November 2019 and May 2022 at four Swiss children’s hospitals. The care follow-up and bereavement follow-up assessments were 330 and 300 days, respectively. We measured out-of-pocket non-medical expenses, individual full-time equivalent units, and personal income, as well as sick leave and vacation days taken. Analyses included descriptive statistics and two-way linear fixed-effects regressions. The analysis included 110 parents (mothers n = 59, fathers n = 51) of 61 children. Children were hospitalized for a median of 7 days (interquartile range 0–21, range 0–227). The fixed-effects models found a positive association between hospitalization, i.e., length of stay, and travel and accommodation expenses (coefficient 4.18, 95% confidence interval 2.20–6.16). On average, for each week of hospitalization, parents spent an additional 29 Swiss francs on travel and accommodation. During the 300-day bereavement follow-up, six (26%) of 23 parents increased their work commitments, while one reported a decrease. Conclusions: Families incur higher travel and accommodation expenses during hospitalization than during non-hospitalized periods. Instrumental support, e.g., parking vouchers, can help families minimize these costs. Future studies should investigate whether early return to work during bereavement is driven by economic considerations or a desire for distraction. Clinical trial registration: Data analyzed in this study were collected as part of a clinical trial, registered on ClinicalTrials.gov, No. NCT04236180, 15 March 2019 What Is Known: • Families of children with life-limiting conditions are at risk of substantial financial burden from high out-of-pocket medical expenses. • It is also known that parents often have to incur out-of-pocket non-medical expenses and reduce their work commitments. Little is known about the economic consequences of losing a child to a life-limiting condition. What Is New: • We provide new longitudinal evidence on the hospitalization- and death-related financial and employment implications for families of children with life-limiting conditions. • Child hospitalizations add to families’ financial burden through increased travel and accommodation expenses. Work commitments rose during early bereavement. Supplementary Information The online version contains supplementary material available at 10.1007/s00431-024-05680-7.


Introduction
Parenting a child with a life-limiting condition (LLC), i.e., conditions "for which there is no reasonable hope of cure and from which children will die" [1, p.10], has an enormous impact on the life of these children's parents, adversely affecting their physical, psychological, emotional, and social health [2][3][4][5][6].In addition, these parents are likely to experience substantial economic adversity [7].Previous research suggests that financial burden is driven by high out-of-pocket (OOP) medical spending, i.e., deductibles and co-payments [8][9][10].However, the costs of taking care of a child with a LLC go beyond medical costs: parents often face OOP nonmedical expenses and employment-related income losses [11][12][13].For instance, Canadian studies revealed that following a child's cancer diagnosis, 64% of mothers and 16% of fathers left their jobs [14] and that illness-related travel and food expenses accounted for nearly three quarters of families' OOP non-medical expenses [15].
Over the course of a LLC, parents have to navigate a range of challenging and traumatic events, including unstable phases of illness, hospital admissions, and even their child's death, any of which can increase their psychological, emotional, social, and financial burdens [2][3][4][5][6][16][17][18][19].For example, families may face extra expenses for traveling to and staying overnight at the hospital.Repeated and lengthy hospitalizations are common for children with LLCs [20,21].However, few studies have examined the effects of a child's hospitalization on OOP non-medical expenses and parental employment [9,[22][23][24][25][26].None of these studies is both LLC-specific and longitudinal [9,[22][23][24][25][26].Having LLCspecific evidence is important since the economic impact is likely accentuated in this population because of higher healthcare resource utilization (e.g., repeated and lengthy hospitalizations) and the manifestation of financial burden over time.
The same is true for evidence on the financial and employment implications of a child's death.Losing a child is a traumatic, life-changing event [27][28][29][30][31]. Conducting research during early bereavement is challenging, as parents are navigating an unimaginably difficult period of their lives [32].As a result, studies about the economic consequences of grief are largely absent from the literature.Nevertheless, the financial and employment implications of losing a child to a LLC can be considerable [33].
For this study, we investigated the financial and employment implications of both hospitalization and bereavement.More specifically, the study's first aim was to explore how a child's hospitalization influences the families' OOP non-medical expenses as well as the parents' income and employment over a 330-day period.Its second aim was to assess changes in parental income and employment over the first 300 days of bereavement.

Methods and materials
This cohort study is a secondary analysis of a prospectively collected panel dataset.The dataset was collected through the "Specialised Paediatric PAlliativE CaRe: Assessing family, healthcare professional and health system outcomes in a multi-site context of various care settings (SPhAERA)" study, which aimed to evaluate the effectiveness of a Swiss specialized pediatric palliative care (SPPC) program [34].The study was conducted between November 2019 and May 2023 with the approval of the responsible Swiss ethics committees (BASEC-Nr.2019-01170) and is registered in ClinicalTrials.gov(NCT04236180).Further information regarding the SPhAERA study can be found elsewhere [34].

Participants and recruitment
This study's population consisted of children with a LLC and their parents (mothers and/or fathers).To be included for study participation, children had to be 0-18 years of age and their families had to be proficient in either German or French.Children whose life expectancy was < 48 h and neonates with medical complications due to prematurity and/or birth complications treated in a neonatal intensive care unit were excluded.Children subject to child protection regulations and their parents were ineligible for study participation.
Participants were recruited between November 2019 and May 2022 at four Swiss study centers, i.e., four (university) children's hospitals.Potential participants were screened for eligibility by the leading medical professional in collaboration with the study team.In one study center, participants were recruited consecutively when they entered the local SPPC program.In the other three study centers, participants potentially in need of SPPC were convenience recruited by the responsible medical professional.Parents provided written informed consent.All families in the study had mandatory health insurance, largely shielding them from OOP medical expenses.

Data collection
The study duration was 330 days including nine assessment timepoints.In the event of a child's death during this period, parents remained enrolled for an additional 300 days (two assessments), starting from the date of death.Diagnostic information and healthcare resource utilization data were extracted from routine data via chart reviews at baseline (day 0) and at eight care follow-up assessments.Healthcare resource utilization data collected at days 15 and 30 were merged to be in line with the collection of economic data, which started at day 30.See Table 1 for a detailed overview of assessment timepoints and variables.Parent and family characteristics and economic data were collected via pseudonymized paper-pencil self-report questionnaires, either distributed in hospital or sent via mail to families' homes.

Exposure variable
To explore associations between hospitalization and family economic outcomes, we used child hospital length of stay (LoS, days) as our exposure measure.For each assessment period, the number of days a child was hospitalized in one of the participating (university) children's hospitals was recorded.

Outcome variables
In addition to a range of OOP non-medical expenses, outcome variables included individual full-time equivalent (FTE) units, personal income, and work absences, i.e., sick leave and vacation days (Table 1).While work absenteeism may not have immediate financial implications, it potentially hampers parents' long-term career perspectives [35].Families' OOP non-medical expenses included illness-related expenses for home healthcare supplies, travel and accommodation, childcare and home help, and special and extraordinary purchases, e.g., home modifications.To measure work commitment and income loss, each parent's FTE unit and income were recorded at study start.At each care followup assessment, parents who had experienced work and/or income-related changes were asked to provide their new FTE unit and/or income.Work absences (both paid and unpaid) of employed parents included two variables: sick leave days and vacation days.In the bereavement follow-up, individual FTE units, income, and work absenteeism, including compassionate days, were measured.OOP non-medical expenses and income data were collected using ordinal categories.All other variables were collected using free text fields.All expenses and income were measured in Swiss francs (CHF).

Other variables
We collected data on a number of relevant diagnostic and socio-demographic/economic characteristics (Table 2).We also assessed the number of hospitalizations and the amount of financial support families received, including support from all sources such as government, charities, and relatives.

Statistical analyses
Descriptive statistics were used to provide an overview of parent, family, and child characteristics, to report on the financial support families received and to describe financial and employment implications.Missing data were analyzed using Little's test [36].
To enhance our understanding of how changes in hospitalization (e.g., spending an additional day in hospital) influences families' economic outcomes, we used two-way linear fixed-effects models [37,38].Our hypothesis was that periods with hospitalization are more costly for families due to increased cost (e.g., travel and transport), but may also be lower due to lower need for other purchases (e.g., home healthcare supplies).The empirical model is specified in Supplemental Material 1, where additional explanations are also provided.
All analyses were performed using the R statistical software (version 4.1.2)[39].The regression analyses were performed using the R plm package (version 2.6-2) [40].A p-value of < 0.05 was applied.

Robustness check
We checked the robustness of our findings via randomeffects, complete-case, and subgroup analyses.Fixed-and random-effects models were compared using the Hausman test (Supplemental Table 1) [41].Additional details regarding the robustness check can be found in Supplementary Material 1.

Study participants
The inclusion criteria were met by 160 of 280 screened families.Of these 160 families, 70 consented for study participation.A family of twins was counted twice because both children participated in the study.Parents who did not complete an assessment at day 30 for reasons other than a child's death were excluded.Overall, the sample utilized in this study's analyses consisted of 110 parents of 61 children with LLCs (Fig. 1).Parent, family, and child characteristics are presented in Table 2.Among parents with foreign citizenship, 12 (48%) were German.The majority of parents not in employment at study entry were mothers (n = 13, 87%).Fifty-three families (87%) had supplementary insurance in addition to compulsory health insurance.

Hospitalizations and LoS
Over the full study period, the median number of hospitalizations per child was 1 (interquartile range (IQR) 0-3, range (Rng.)0-6).For the 30-day and 90-day assessment periods, the maximum number of hospitalizations per child was 2 and 3, respectively.Seventeen children (28%) had no hospitalization.The median total hospital LoS was 7 days (IQR 0-21, Rng.0-227).Details on child hospital LoS are provided in Fig. 2.

OOP non-medical expenses and financial support
Figures 3A-D and 4A-E detail the outcomes of the 330day follow-up.The proportion of families that did not incur any travel and accommodation expenses peaked at day 120 (Fig. 3B)-the same day the proportion of children hospitalized was lowest.Childcare and home help expenses averaged CHF480 per month for the 20 families (33%) that had such expenses (Fig. 3C).Throughout study participation, six families (10%) spent > CHF10,000 for special and extraordinary purchases (Fig. 3D).At each assessment, 50% or fewer families had financial support (Supplemental Table 5).

Employment outcomes
The highest share of parents (n = 9, 8%) decreasing their individual FTE, primarily mothers (n = 7, 78%), was observed on day 30 (Fig. 4B).Throughout the 330-day follow-up, an average of 33% of mothers were not in employment, while for fathers, this applied to 5%.

Missing data
The amount of missing data was highest for sick leave and vacation days (up to 21%) and lowest for OOP non-medical expenses (5% max.) (Supplemental Table 3).As for the latter, Little's test showed that missing data were likely to be completely random (Supplemental Table 4).

Hospitalization-related implications
The seven care follow-up assessments provided 344 family and 633 parent observations for use in the regression analyses.The adjusted fixed-effects analyses showed a positive association of child hospital LoS with travel and accommodation expenses (coefficient 4.18, 95% confidence interval 2.20-6.16).For every 1-day increase in hospital LoS, travel and accommodation expenses increased by 6.2% compared to the reference group.The median hospital LoS was 7 days (IQR 0-21, Rng.0-227).With a mean difference in travel and accommodation expenses of CHF4.18, families spent an additional CHF29 for every week of their child's hospitalization.No other associations were found between child hospital LoS and our other outcomes (Table 3).

Robustness check
The complete-case and subgroup analyses support the findings of our fixed-effects analyses, with two exceptions: Hospital LoS was no longer associated with travel and accommodation expenses for families with a household income of ≥ CHF100,000 and those with a home-to-hospital travel distance of ≤ 20 km (Supplemental Tables 7-12).

Discussion
In a sample of parents of children with a LLC, we investigated the financial and employment implications of two events: child hospitalization and death.Child hospitalization, i.e., hospital LoS, was positively associated with families' travel and accommodation expenses.During the first 120 days of bereavement, more than one-fifth of grieving parents increased their work commitments.
The additional travel and accommodation expenses endured by families are likely explained by parents maintaining a bedside presence during their child's hospitalization, e.g., for transportation, parking, and board and lodging [22][23][24]26].In our sample, the extra travel and accommodation expenses incurred by parents per week of hospitalization were rather low.For some parents, settingspecific factors such as parking and food vouchers or freeof-charge hospital accommodation for parents may have limited these expenses.However, in settings with less support, these expenses could be much higher.Moreover, with repeated and lengthy hospitalizations, travel and accommodation expenses are likely to be greater.
Consistent with previous research, our analyses indicate that families residing farther away from the hospital experience increased travel and accommodation expenses [22,24].Families with lower income may have reported their spending more accurately, as they were required to provide detailed records to financial assistance programs.This may explain the association of hospital LoS with travel and accommodation expenses for these families.Another explanation could be that, due to affordable housing options, families with lower income live farther away from hospitals.
There are several possible explanations for the lack of association between hospital LoS and other OOP non-medical expenses.First, during their child's hospitalization, a family may purchase home healthcare supplies in preparation for their child's hospital-to-home transition.Being prepared in terms of medication, equipment, and supplies has been shown to be a priority for families [42].Second, informal care provided by relatives and friends may explain both the low number of families that incurred formal childcare and home help expenses and the lack of association between such expenses and hospital LoS.Here, our findings contrast with previous research, which suggested that childcare expenses, e.g., for siblings, would normally increase during hospitalizations [22,23].Cultural differences may have an influence on families' informal caregiving arrangements [43].Third, a child's functional limitations, namely, their dependency on medical technology and equipment, may be a stronger explanatory factor for increased special and Fig. 1 Flow diagram of study participants.The number of families is equal to the number of children; one family of twins was counted twice.Parents/families who did not complete assessments at day 30 or participate in the bereavement follow-up were excluded.*Only one parent (mother or father) was excluded/dropped-out ◂ Fig. 2 Child hospital LoS.Assessment periods were 30 days each for the first five assessments and 90 days each for the last two.The line gives the mean child hospital LoS.Each bar indicates the proportion of children hospitalized for at least 1 day for the corresponding assessment extraordinary expenses than hospitalization [12].Although few families in our study incurred such expenses, for those who did, they were exceedingly high.
Contrary to our expectations, we did not find any significant hospitalization-related employment or income implications.Income replacement mechanisms, e.g., paid sick leave, and flexible employment arrangements, e.g., working from home, may have protected families from adverse effects.Previous research suggests that income loss is less severe in parents with flexible work arrangements [23].As a contextual matter, it is worth noting that data collection occurred during the coronavirus disease 2019 pandemic, which brought a substantial increase in flexible work practices [44].
In addition, families were recruited a median of 1 year after their child's diagnosis, when certain employment adaptations may already have taken place.For instance, mothers of children diagnosed during maternity leave may have extended that leave.A previous study of employment implications in families of children with special healthcare needs found that forgone employment was disproportionately high among mothers of young children (0-5 years) [45].Compared to mothers in the general population, those in our study were less likely to be employed.Across the care follow-up assessments, an average of 33% of participating mothers did not engage in employment, compared with 17% in the general population [46].
Regarding the bereavement follow-up, parents' increasing work commitments may have been motived by both economic considerations and a desire to resume work, e.g., as a means of distraction [33,47].That is, the observed increases in work commitment do not necessarily indicate full readiness or restored functionality [33].In general pediatrics, parental grief-related costs associated with work-presenteeism outweighed those associated with work-absenteeism [33].The same principle may also explain a number of our bereavement follow-up observations of parental income, sick leave, and vacation days.One of this study's major strengths is its longitudinal design: multiple assessments provided us with a realistic representation of how LLC-related events-and costsdevelop over time.Another is its fixed-effects approach, which controls for time-invariant heterogeneity and confounders.Nevertheless, it also has notable limitations, including the relatively small sample size and limited number of hospitalizations, which limit statistical power and generalizability.In addition, sample bias cannot be ruled out, as families experiencing high psychological,  emotional, social, or financial burdens may have been more likely to decline study participation.This effect might have been accentuated by additional attrition bias during the study.Moreover, outcome data were selfreported, leaving them vulnerable to response and recall bias.Some parents may have been reluctant to disclose their income.However, our instruments' discrete response categories and primarily monthly assessments would have minimized this type of bias.Finally, hospitalization rates may have been influenced by the coronavirus disease 2019 pandemic.

Conclusions
Hospitalization-related travel and accommodation expenses exacerbate the often substantial financial burden experienced by families of children with LLCs.To protect these families' financial well-being, adequate financial support should be made available to them.Where such financial support currently exists, additional efforts should be made both to enhance affected parents' awareness and assist them with the application processes.Future research is also needed to further explore and explain the effect of LLCs in children on their parents' employment, e.g., by comparing families of children with a LLC to those of children without a LLC.To date, little is known of the factors behind these parents' decisions to reduce or increase their work commitments through their children's illness and their own bereavement.

Fig. 3 A
Fig. 3 A-D Family OOP expenses.Assessment periods were 30 days each for the first five assessments and 90 days each for the last two.In subfigures A and B, the sum of the proportions is less than 100% due to missing data.CHF indicates Swiss francs

Fig. 4 A 3
Fig.4A-E Parental FTE, income, and work absences.Assessment periods were 30 days each for the first five assessments and 90 days each for the last two.The sum of the proportions is less than 100%

Table 1
Overview of assessment timepoints and variables a Baseline assessment b Days after the child's date of death

Table 2
Parent, family, and child characteristics SD standard deviation, IQR inter quartile range, CHF Swiss francs a A family (two parents) of twins was counted twice because both children had an LLC and participated in the study Illness duration gives the number of days between the date of diagnosis of the LLC and the date of study entry [48]mber of children includes the child with an LLC c Gross annual household income, Swiss average lies at CHF157,008[48]d